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RecruitingNCT04003922Updated Jan 8, 2024

Study on Patients With Rasmussen Encephalitis Treated With Adalimumab: Efficacy and Tolerance in the Short and Long Term

An observational study in Rasmussen Encephalitis, sponsored by Assistance Publique Hopitaux De Marseille. Recruiting at 16 sites in France. Open to participants aged 2 Years and older. Per ClinicalTrials.gov, last updated 2024-01-08.

Sponsored by Assistance Publique Hopitaux De Marseille · Observational

Study type
Observational
Model
Other
Time perspective
Other
Enrollment
40
Ages
2 Years and older
Sex
All
01

Study summary

Since the initial study of cases reported between 2009 and 2015, adalimumab has become in France the immunomodulatory reference treatment used after failure of corticosteroids and immunoglobulins before a possible recourse to the hemispherotomy. This observational study is intended to document the long-term efficacy and safety of Adalimumab therapy in patients with Rasmussen encephalitis.

Read the detailed description

Rasmussen Encephalitis (ER) is a particularly severe chronic inflammatory brain disorder resulting in the progressive destruction of a hemisphere. It is a rare disease although at present no precise prevalence is available. It begins preferentially in children.

This inflammatory process is accompanied by a progressive loss of function of the affected hemisphere, associated with a pharmaco-resistant partial epilepsy. The diagnosis is based on a bundle of clinical, radiological and electroencephalographic arguments. CSF analysis directs diagnosis in 50% of cases.

No anti-epileptic treatment can stop seizures. Only hemispherotomy (surgical disconnection of a cerebral hemisphere) allows it but it is associated with definitive motor and cognitive deficits.

Over the last 20 years, new therapeutic trials have focused on immuno-modulatory treatments targeted at the T-lymphocyte pathway, including tacrolimus. Although they seem to be more effective than immunoglobulins or corticosteroids, it remains transient. In addition, the number of published cases is low. In this context, starting in 2009, it has been proposed to use adalimumab (Ab anti TNF) based on:

  • 1 / study of a case index
  • 2 / knowledge of the pathophysiology of Rasmussen Encephalitis. To date, very few data provide precise information on the efficacy or tolerance of the use of this product in the longer term. This information is essential to confirm the place of adalimumab in the therapeutic arsenal against Rasmussen encephalitis.

Thus, in the continuation of the work carried out previously (French study on the cases between 2009 and 2015), the aim of this research projet is to complete the follow-up of the patients who previously took part in in the first study and to establish the follow-up of the patients treated by adalimumab since then.

02

Conditions studied

  • Rasmussen Encephalitis
03

Who can participate

Ages eligible
2 Years and older
Sexes eligible
All
Accepts healthy volunteers
No
Sampling method
Probability sample

Study population

The implementation of a treatment with adalimumab in a patient with an ER requires the performance of a CPR within a reference center Epilepsies Rares if one complies with the rules of good clinical practice. Thus, all ER patients treated with adalimumab are referenced in a referral center. There are 7 reference centers in France, they are all partners of the study.

Inclusion criteria

  • Confirmed diagnosis of Rasmussen Encephalitis
  • Patient aged more than 2 years
  • Compliant treatment with adalimumab

Exclusion criteria

Exclusion Criteria:

  • Patient with a differential diagnosis
  • Patient suffering from Rassmussen Encephalitis but not treated with adalimumab
  • Patient who has not signed the informed consent
04

Study design

Observational model
Other
Time perspective
Other
Enrollment
40 participants (estimated)
Target follow-up
5 Years
Patient registry
Yes

Interventions

  • OtherAdalimumab treatment

    Retrospective observational study initially and then prospective with inclusion of all the patients carrying an ER treated by Adalimumab in France. Follow-up will continue for one year if adalimumab is discontinued.

05

What researchers measure

Primary outcomes

  1. Efficiency of the treatment measured by the reduction of the number of epileptic seizures

    The change of epileptic seizures (Will be considered as a responder patient any patient with a 50% decrease in the number of seizures between the time of inclusion and 6 months after the start of treatment). This response to treatment is re-evaluated at 12 months then every 6 months for 5 years.

    Time frame: 5 years

  2. Efficiency of the treatment measured by the stability if the cognitive assessment

    Stability of the cognitive assessment: variation of less than 10 points on each of the different composite indices (QIT, ICV, IRF, IMT, IVT) of age-appropriate Weschler scales, between two cognitive assessments of 6 months.

    Time frame: 6 months

06

Study locations

1 of 16 sites recruiting
  • Chu de Bordeaux
    Bordeaux, France
    • Frédéric Villega · Contact
    Not yet recruiting
  • Chu Brest
    Brest, France
    • Jérémie Lefranc · Contact
    Not yet recruiting
  • Chru de Lille
    Lille, France
    • Sylvie Nguyen · Contact
    Not yet recruiting
  • Chu Limoges
    Limoges, France
    • Cécile Laroche · Contact
    Not yet recruiting
  • Hospices Civils de Lyon
    Lyon, France
    • Dorothée Ville · Contact
    Not yet recruiting
  • Service Neuropédiatrie
    Marseille, 13005, France
    • Anne Lepine · Contact
    Recruiting
  • Chu Montpellier
    Montpellier, France
    • Agathe Roubertie · Contact
    Not yet recruiting
  • Chu de Nancy
    Nancy, France
    • Louis Maillard · Contact
    Not yet recruiting
  • Chu Necker Ap-Hp
    Paris, France
    • Rima Nabbout · Contact
    Not yet recruiting
  • Chu Pitie Salpietriere Ap-Hp
    Paris, France
    • Sophie Dupont · Contact
    Not yet recruiting
  • Chu Robert Debre
    Paris, France
    • Stéphane Auvin · Contact
    Not yet recruiting
  • Fondation Adolphe de Rothshild
    Paris, France
    • Christine Bulteau · Contact
    Not yet recruiting
  • Chu de Rennes
    Rennes, France
    • Silvia Napuri · Contact
    Not yet recruiting
  • Chu Strasbourg
    Strasbourg, France
    • Anne De Saint-Martin · Contact
    Not yet recruiting
  • Chu Toulouse
    Toulouse, France
    • Claude Cances · Contact
    Not yet recruiting
  • Chru de Tours
    Tours, France
    • Maximilien Perivier · Contact
    Not yet recruiting
07

Registry details

Key details

Study ID
NCT04003922
Lead sponsor
Assistance Publique Hopitaux De Marseille
Responsible party
Sponsor
First posted
Jul 1, 2019
Start date
Apr 21, 2020
Primary completion
Apr 2024 (estimated)
Completion
Oct 2025 (estimated)
Last update
Jan 8, 2024

Study contacts

Anne Lépine
Contact
anne.lepine@ap-hm.fr
0491387267 ext. 33
DRS AP-HM
Contact
drci@ap-hm.fr
0491381499 ext. 33
Jean-Olivier ARNAUD
study director · Assistance Publique- Hôpitaux de Marseille

Oversight

Data monitoring committee
No
FDA-regulated drug
No
FDA-regulated device
No
View the source record on ClinicalTrials.gov ↗

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